Purpose:The purpose of this study was to evaluate the clinical outcome of patients with refractory neurotrophic keratopathy (NK) in stages 2 and 3 treated with topical insulin.Methods:Retrospective analysis of eyes with NK in stages 2 and 3 refractory to standard medical and/or surgical treatment which were treated with topical insulin (1 unit per mL). This treatment was applied 4 times per day and was continued until the persistent epithelial defect (PED) or ulcer resolved. The primary outcome of the study was the complete reepithelialization of the PED or persistent ulcer. “Best-corrected visual acuity” pretreatment and posttreatment, “days until complete reepithelialization” data, and anterior segment photographs were obtained. Outcome measures were compared before and after treatment in both groups using paired and independent samples t tests.Results:Twenty-one eyes were included in this study, and 90% achieved complete reepithelialization of the PED and/or persistent ulcer within 7 to 45 days of follow-up. The mean number of days until complete reepithelialization was significantly lower in NK stage 2 (18 ± 9 days) when compared with NK stage 3 (29 ± 11 days) (P = 0.025). The best-corrected visual acuity improved significantly in both NK stage 2 (P < 0.001) and NK stage 3 (P = 0.004). No side effects were reported during the follow-up.Conclusions:Our results suggest that topical insulin drops may be an effective therapeutic in refractory NK. This therapy may prove extremely useful because of its low cost and high accessibility.
Horner syndrome (HS) is characterized by pupillary miosis, upper eyelid ptosis, apparent enophthalmos and facial anhidrosis. It has been reported as a rare complication of thoracic surgery. We describe a 12-year-old girl who presented with a pre-ganglionic HS one day after cardiothoracic surgery for post-ductal coarctation of the aorta. Apraclonidine test was positive on the twelfth day following the onset of HS.
PurposeTo evaluate the clinical progression of patients with refractory Neurotrophic Keratopathy (NK) treated with topical insulin eye drops and its efficacy in treating NK in stages 2 and 3.MethodsQuasi‐Experimental, randomized study, from October 2018 to December 2019, of 10 patients (11 eyes—13 to 92 years) with NK in stages 2 and 3. Patients included were refractory to standard medical and/or surgical treatment. Drops were applied 3‐4 times a day and a bandage contact lens was applied. Close follow‐up of patients was assured (daily in week one and weekly thereafter) and anterior segment photos were taken on each visit. Insulin eye drops were continued until the NK persistent epithelial defect or ulcer resolved. Treatment would be discontinued if the condition did not improve or worsened.ResultsTen out of eleven eyes had complete resolution of the NK persistent epithelial defect and/or ulcer within 12‐45 days of follow‐up. One of the patients’ epithelial defect improved significantly although the wound did not fully heal within 42 days of treatment. No side‐effects were reported during the full extent of the treatment.ConclusionsAlthough we recognize some limitations to our study (uncontrolled series of patients), our results suggest that topical insulin drops could be an effective treatment in refractory NK. Furthermore, this therapy may prove extremely useful due to its low cost and high accessibility. However, more studies are needed to evaluate the efficacy, posology, duration and side‐effects of this treatment.
PurposeWe aim to report a case of a 15 year‐old patient with severe and rapid bilateral visual impairment due to progressive cone dystrophy and describe the associated mutations.MethodsThis is a case report of a patient with a rare disease who underwent full ophthalmologic evaluations including optical coherence tomography (OCT) of the posterior pole, fundus autofluorescence (FAF) and electroretinography (ERG).ResultsA 15‐year‐old girl was presented at our Department with progressive vision loss and poor colour vision complaints. The patient reported these complaints for a year, despite a full uneventful ophthalmic evaluation at that time. Best‐corrected visual acuity was 6/60 for each eye and Ishihara 24‐plates colour test was altered. Fundoscopic evaluation showed a maculopathy with spotty pigment changes in addition to temporal pallor of the optic disk. Posterior pole OCT showed a reduced outer nuclear layer and retinal pigment epithelium unspecific changes. FAF revealed a central dark area surrounded by a ring of increased autofluorescence. ERG was inconclusive due to poor collaboration, although it was hypothesised to have a slight cone dysfunction. Moreover, genetic analyses of ABCA4 and CDHR1 genes were found to be positive for multiple mutations. The c.6816 + 2T>A (p.Leu2035Pro) mutation was never described and the c.6104T>C (p.Leu2035Pro) mutation was only present in one patient with Stargardt disease. After a period of 2 years of follow‐up visual function and retinal disease remained stable.ConclusionsProgressive cone dystrophy is a rare inherited ocular disorder characterized by the loss of cone cells. This case report emphasizes the need to reach a clear diagnosis when uncommon symptoms appear in an otherwise normal ophthalmic evaluation and also describe newer mutations in ABCA4 gene enhancing our knowledge about this disease.
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